Abstract
Purpose
Osteogenesis imperfecta (OI) is a genetic disorder responsible for various symptoms including deformities and frequent fractures. Bone allografting is poorly documented in this condition. The objective of this study was to describe our experience and assessments in a consecutive series of OI patients.
Methods
Thirty-nine lower limb allograft procedures (28 femurs, 11 tibias) were performed in 26OI patients (mean age, 12.9 years). They were classified as type III of Sillence (17), type IV (6), and 3 recessive forms. The indications for surgery were correction of deformity (19), fracture (16), and non-union (4). In all cases, bone allografting was added to reinforce areas of fragility and in 28 cases for osteosynthesis to lock the rotations at the osteotomy site and to avoid screwed metallic plate. The duration of bone consolidation and allograft fusion was assessed. Complications and Gillette functional score were reported.
Results
The mean follow-up was 6.7years (range, 2 to 10 years). On average, bone consolidation was achieved after 3.3 months and graft fusion after 7.7 months. No bone allograft-related complications were observed and there was any secondary displacement. The Gillette functional score was improved in 23 patients and stable in three cases. Complications were reported in two cases: one partial allograft resorption and one delayed consolidation of a non-union. One refracture was observed but after a significant trauma in a child who had regained significant physical activity.
Conclusions
Bone allografting in children with OI is a reliable method of biological fixation, allowing efficient fusion and contributing to increased bone capital and functional outcome.
Similar content being viewed by others
Data availability
All data are available on request from the corresponding author and transmitted as an anonymized Excel file.
Code availability
Not applicable.
References
Kuurila K, Kaitila I, Johansson R, Grénman R (2002) Hearing loss in Finnish adults with osteogenesis imperfecta: a nationwide survey. Ann Otol Rhinol Laryngol 111:939–946
Alanay Y, Avaygan H, Camacho N, Utine GE, Boduroglu K, Aktas D et al (2010) Mutations in the gene encoding the RER protein FKBP65 cause autosomal-recessive osteogenesis imperfecta. Am J Hum Genet 86:551–559
Sillence DO, Senn A, Danks DM (1979) Genetic heterogeneity in osteogenesis imperfecta. J Med Genet 16(2):101–116
Doyard M, Bacrot S, Huber C, Di Rocco M et al (2018) FAM46A mutations are responsible for autosomal recessive osteogenesis imperfecta. J Med Genet 55(4):278–284
Mortier GR, Cohn DH, Cormier-Daire V, Hall C, Krakow D, Mundlos S et al (2019) Nosology and classification of genetic skeletal disorders: 2019 revision. Am J Med Genet A 179(12):2393–2419
Zeitlin L, Fassier F, Glorieux FH (2003) Modern approach to children with osteogenesis imperfecta. J Pediatr Orthop 10:77–87
Glorieux FH (2007) Experience with bisphosphonates in osteogenesis imperfecta. Pediatrics. 119(S 2):S163–S165
Gamble JG, Strudwick WJ, Rinsky LA, Bleck EE (1988) Complications of intramedullary rods in osteogenesis imperfecta: Bailey-Dubow rods versus non elongating rods. J Pediatr Orthop 8(6):645–649
Esposito P (2008) Plotkin H. Surgical treatment of osteogenesis imperfecta: current concepts. Curr Opin Pediatr 20(1):52–57
Zeitlin L, Fassier F, Glorieux FH (2003) Modern approach to children with osteogenesis imperfecta. J Pediatr Orthop B 12:77–87
Enright WJ, Noonan KJ (2006) Bone plating in patients with type III osteogenesis imperfecta: Results and complications. Iowa Orthop J 26:37–40
Springer BD, Berry DJ, Lewallen DG (2003) Treatment of periprosthetic femoral fractures following total hip arthroplasty with femoral component revision. J Bone Joint Surg Am 85:2156–2162
Wang JW, Weng LH (2003) Treatment of distal femoral nonunion with internal fixation, cortical allograft struts and autogenous bone-grafting. J Bone Joint Surg Am 85:436–440
Puvanesarajah V, Shapiro JR, Sponseller PD (2015) Sandwich allografts for long-bone nonunions in patients with osteogenesis imperfecta: a retrospective study. J Bone Joint Surg Am 97(4):318–325. https://doi.org/10.2106/JBJS.N.00584
Lin D, Zhai W, Lian K, Ding Z (2013) Results of a bone splint technique for the treatment of lower limb deformities in children with type I osteogenesis imperfecta. Indian J Orthop 47(4):377–381. https://doi.org/10.4103/0019-5413.114922
Novacheck TF, Stout JL, Tervo R (2000) Reliability and validity of the Gillette Functional Assessment Questionnaire as an outcome measure in children with walking disabilities. J Pediatr Orthop 20:75–81
Whelan DB, Bhandari M, Stephen D, Kreder H, McKee MD, Zdero R, Schemitsch EH (2010) Development of the radiographic union score for tibial fractures for the assessment of tibial fracture healing after intramedullary fixation. J Trauma 68(3):629–632
Song MH, Yoo SH, Kang SW, Kim YJ, Park GT, Pyeun YS (2015) Coronal alignment of the lower limb and the incidence of constitutional varus knee in Korean females. Knee Surg Relat Res 27(1):49–55
Zlowodzki M, Brink O, Switzer J, Wingerter S, Woodall J Jr, Petrisor BA, Kregor PJ, Bruinsma DR, Bhandari M (2008) The effect of shortening and varus collapse of the femoral neck on function after fixation of intracapsular fracture of the hip: a multi-centre cohort study. J Bone Joint Surg Br 90(11):1487–1494. https://doi.org/10.1302/0301-620X.90B11.20582
Lascombes P (2010) Flexible intramedullary nailing. The Nancy University Manual. Springer-Verlag, Berlin, Heidelberg, p 317
Langlais T, Pannier S, De Tienda M, Dukan R, Finidori G, Glorion C, Péjin Z (2021) ‘In-Out-In’ K-wires sliding in severe tibial deformities of osteogenesis imperfecta: a technical note. J Pediatr Orthop B 30(3):257–263. https://doi.org/10.1097/BPB.0000000000000785
Franzone JM, Rogers KJ, Kruse RW (2018) Categorization of the usage of adjunctive structural allograft bone graft in extremity surgery for patients with osteogenesis imperfecta. J Long Term Eff Med Implants 28(3):205–208
Mueller B, Engelbert R, Baratta-Ziska F, Bartels B, Blanc N et al (2018) Consensus statement on physical rehabilitation in children and adolescents with osteogenesis imperfecta. Orphanet J Rare Dis 13(1):158. https://doi.org/10.1186/s13023-018-0905-4
Montpetit K, Lafrance ME, Glorieux FH, Fassier F, Hamdy R, Rauch F (2021) Predicting ambulatory function at skeletal maturity in children with moderate to severe osteogenesis imperfecta. Eur J Pediatr 180(1):233–239. https://doi.org/10.1007/s00431-020-03754-w
Cho T-J, Lee K, Chang-Wug O, Park MS, Yoo WJ, Choi IH (2015) Locking plate placement with unicortical screw fixation adjunctive to intramedullary rodding in long bones of patients with osteogenesis imperfecta. J Bone Joint Surg Am 97(9):733–737. https://doi.org/10.2106/JBJS.N.01185
Vlad C, Georgescu I, Gavriliu TS, Hodorogea DI, Nayef E (2012) Burnei'sprocedure inthe treatment of longbonepseudarthrosis in patients having osteogenesisimperfecta or congenitalpseudarthrosis of tibia - preliminaryreport. J Med Life 5(2):215–221
Devalia KL, Mehta R, Yagnik MG (2005) Use of maternal bone grafting for long standing segmental gap non-union in Osteogenesis Imperfecta: a case report with review of literature. Injury 36(9):1130–1134. https://doi.org/10.1016/j.injury.2004.11.018
Nguyen H, Morgan DAF, Forwood MR (2007) Sterilization of allograft bone: effects of gamma irradiation on allograft biology and biomechanics. Cell Tissue Bank 8(2):93–105. https://doi.org/10.1007/s10561-006-9020-1
Author information
Authors and Affiliations
Contributions
Mathilde Gaume: literature research, clinical studies, data analysis, statistical analysis, manuscript preparation, manuscript editing; Sarah El Yahiaouni: literature research, data analysis, manuscript preparation; Marine De Tienda: clinical studies, manuscript editing; Genevieve Baujat: clinical studies, manuscript editing; Valérie Cormier-Daire: manuscript editing; Valérie Dumaine: manuscript editing; Stéphanie Pannier: manuscript editing; Georges Finidori: guarantor of integrity of the entire study, study concepts and design, clinical studies, manuscript editing; Zagorka Pejin: guarantor of integrity of the entire study, study concepts and design, clinical studies, manuscript editing.
Corresponding author
Ethics declarations
Ethical approval
The Ethics Committee of our institution approved this study (IRB registration: #00011928).
Informed consent
All parents or legal guardians provided their informed consent.
Consent to participate
All patients consented to participate.
Consent to publish
All authors consent to publish the present study in case of acceptance.
Conflict of interest
The authors declare no competing interests.
Guarantor
The scientific guarantor of this publication is Dr Mathilde GAUME.
Statistics and biometry
One of the authors has significant statistical expertise.
Study subjects or cohorts overlap
None of the study subjects or cohorts have been previously reported.
Methodology
Retrospective and performed at one institution.
Additional information
Publisher’s note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
Gaume, M., El Yahiaouni, S., De Tienda, M. et al. Bone allografting: an original method for biological osteosynthesis and bone reinforcement in children with osteogenesis imperfecta. International Orthopaedics (SICOT) 47, 1863–1869 (2023). https://doi.org/10.1007/s00264-023-05818-6
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00264-023-05818-6